Zebrafish motile cilia as a model for primary ciliary dyskinesia

Detalhes bibliográficos
Autor(a) principal: Pinto, Andreia L.
Data de Publicação: 2021
Outros Autores: Rasteiro, Margarida, Bota, Catarina, Pestana, Sara, Sampaio, Pedro, Hogg, Claire, Burgoyne, Thomas, Lopes, Susana S.
Tipo de documento: Artigo
Idioma: eng
Título da fonte: Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
Texto Completo: http://hdl.handle.net/10362/125607
Resumo: Funding Information: Funding: This work was a product of the Project LYSOCIL funded by the European Union Horizon 2020 research and innovation under grant agreement No 811087. It received funding from Fundação para a Ciencia e tecnologia, grant PTDC/BEX-BID/1411/2014; M.R. was funded by the fellowship PD/BD/136927/2018. P.S was funded by the fellowship SFRH/BD/111611/2015; C.B. was funded by the fellowship SFRH/BD/141034/2018; SSL was funded by FCT CEEC-IND 2018. Funding Information: Acknowledgments: The authors want to thank the Fish Facility from NMS. This work was developed with the support of the Fish Facility NMS|FCM that is part of CONGENTO, a Research Infrastructure co-financed by Lisboa Regional Operational Programme (Lisboa2020), under the PORTUGAL 2020 Partnership Agreement, through the European Regional Development Fund (ERDF) and Fundação para a Ciência e Tecnologia (Portugal) LISBOA-01-0145-FEDER-022170. A.P. wants to thank the Royal Brompton and Harefield hospital, part of the Guy’s and St Thomas’ foundation trust, London. Publisher Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland.
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spelling Zebrafish motile cilia as a model for primary ciliary dyskinesiaAnimal modelElectron tomographyLeft–right organizerMotile ciliaOlfactory pitTransmission electron microscopyZebrafishCatalysisMolecular BiologySpectroscopyComputer Science ApplicationsPhysical and Theoretical ChemistryOrganic ChemistryInorganic ChemistrySDG 3 - Good Health and Well-beingFunding Information: Funding: This work was a product of the Project LYSOCIL funded by the European Union Horizon 2020 research and innovation under grant agreement No 811087. It received funding from Fundação para a Ciencia e tecnologia, grant PTDC/BEX-BID/1411/2014; M.R. was funded by the fellowship PD/BD/136927/2018. P.S was funded by the fellowship SFRH/BD/111611/2015; C.B. was funded by the fellowship SFRH/BD/141034/2018; SSL was funded by FCT CEEC-IND 2018. Funding Information: Acknowledgments: The authors want to thank the Fish Facility from NMS. This work was developed with the support of the Fish Facility NMS|FCM that is part of CONGENTO, a Research Infrastructure co-financed by Lisboa Regional Operational Programme (Lisboa2020), under the PORTUGAL 2020 Partnership Agreement, through the European Regional Development Fund (ERDF) and Fundação para a Ciência e Tecnologia (Portugal) LISBOA-01-0145-FEDER-022170. A.P. wants to thank the Royal Brompton and Harefield hospital, part of the Guy’s and St Thomas’ foundation trust, London. Publisher Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland.Zebrafish is a vertebrate teleost widely used in many areas of research. As embryos, they develop quickly and provide unique opportunities for research studies owing to their transparency for at least 48 h post fertilization. Zebrafish have many ciliated organs that include primary cilia as well as motile cilia. Using zebrafish as an animal model helps to better understand human diseases such as Primary Ciliary Dyskinesia (PCD), an autosomal recessive disorder that affects cilia motility, currently associated with more than 50 genes. The aim of this study was to validate zebrafish motile cilia, both in mono and multiciliated cells, as organelles for PCD research. For this purpose, we obtained systematic high-resolution data in both the olfactory pit (OP) and the left–right organizer (LRO), a superficial organ and a deep organ embedded in the tail of the embryo, respectively. For the analysis of their axonemal ciliary structure, we used conventional transmission electron microscopy (TEM) and electron tomography (ET). We characterised the wild-type OP cilia and showed, for the first time in zebrafish, the presence of motile cilia (9 + 2) in the periphery of the pit and the presence of immotile cilia (still 9 + 2), with absent outer dynein arms, in the centre of the pit. In addition, we reported that a central pair of microtubules in the LRO motile cilia is common in zebrafish, contrary to mouse embryos, but it is not observed in all LRO cilia from the same embryo. We further showed that the outer dynein arms of the microtubular doublet of both the OP and LRO cilia are structurally similar in dimensions to the human respiratory cilia at the resolution of TEM and ET. We conclude that zebrafish is a good model organism for PCD research but investigators need to be aware of the specific physical differences to correctly interpret their results.Centro de Estudos de Doenças Crónicas (CEDOC)NOVA Medical School|Faculdade de Ciências Médicas (NMS|FCM)RUNPinto, Andreia L.Rasteiro, MargaridaBota, CatarinaPestana, SaraSampaio, PedroHogg, ClaireBurgoyne, ThomasLopes, Susana S.2021-10-04T23:16:41Z2021-08-022021-08-02T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttp://hdl.handle.net/10362/125607eng1661-6596PURE: 33427504https://doi.org/10.3390/ijms22168361info:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2024-03-11T05:06:29Zoai:run.unl.pt:10362/125607Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-20T03:45:44.190884Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse
dc.title.none.fl_str_mv Zebrafish motile cilia as a model for primary ciliary dyskinesia
title Zebrafish motile cilia as a model for primary ciliary dyskinesia
spellingShingle Zebrafish motile cilia as a model for primary ciliary dyskinesia
Pinto, Andreia L.
Animal model
Electron tomography
Left–right organizer
Motile cilia
Olfactory pit
Transmission electron microscopy
Zebrafish
Catalysis
Molecular Biology
Spectroscopy
Computer Science Applications
Physical and Theoretical Chemistry
Organic Chemistry
Inorganic Chemistry
SDG 3 - Good Health and Well-being
title_short Zebrafish motile cilia as a model for primary ciliary dyskinesia
title_full Zebrafish motile cilia as a model for primary ciliary dyskinesia
title_fullStr Zebrafish motile cilia as a model for primary ciliary dyskinesia
title_full_unstemmed Zebrafish motile cilia as a model for primary ciliary dyskinesia
title_sort Zebrafish motile cilia as a model for primary ciliary dyskinesia
author Pinto, Andreia L.
author_facet Pinto, Andreia L.
Rasteiro, Margarida
Bota, Catarina
Pestana, Sara
Sampaio, Pedro
Hogg, Claire
Burgoyne, Thomas
Lopes, Susana S.
author_role author
author2 Rasteiro, Margarida
Bota, Catarina
Pestana, Sara
Sampaio, Pedro
Hogg, Claire
Burgoyne, Thomas
Lopes, Susana S.
author2_role author
author
author
author
author
author
author
dc.contributor.none.fl_str_mv Centro de Estudos de Doenças Crónicas (CEDOC)
NOVA Medical School|Faculdade de Ciências Médicas (NMS|FCM)
RUN
dc.contributor.author.fl_str_mv Pinto, Andreia L.
Rasteiro, Margarida
Bota, Catarina
Pestana, Sara
Sampaio, Pedro
Hogg, Claire
Burgoyne, Thomas
Lopes, Susana S.
dc.subject.por.fl_str_mv Animal model
Electron tomography
Left–right organizer
Motile cilia
Olfactory pit
Transmission electron microscopy
Zebrafish
Catalysis
Molecular Biology
Spectroscopy
Computer Science Applications
Physical and Theoretical Chemistry
Organic Chemistry
Inorganic Chemistry
SDG 3 - Good Health and Well-being
topic Animal model
Electron tomography
Left–right organizer
Motile cilia
Olfactory pit
Transmission electron microscopy
Zebrafish
Catalysis
Molecular Biology
Spectroscopy
Computer Science Applications
Physical and Theoretical Chemistry
Organic Chemistry
Inorganic Chemistry
SDG 3 - Good Health and Well-being
description Funding Information: Funding: This work was a product of the Project LYSOCIL funded by the European Union Horizon 2020 research and innovation under grant agreement No 811087. It received funding from Fundação para a Ciencia e tecnologia, grant PTDC/BEX-BID/1411/2014; M.R. was funded by the fellowship PD/BD/136927/2018. P.S was funded by the fellowship SFRH/BD/111611/2015; C.B. was funded by the fellowship SFRH/BD/141034/2018; SSL was funded by FCT CEEC-IND 2018. Funding Information: Acknowledgments: The authors want to thank the Fish Facility from NMS. This work was developed with the support of the Fish Facility NMS|FCM that is part of CONGENTO, a Research Infrastructure co-financed by Lisboa Regional Operational Programme (Lisboa2020), under the PORTUGAL 2020 Partnership Agreement, through the European Regional Development Fund (ERDF) and Fundação para a Ciência e Tecnologia (Portugal) LISBOA-01-0145-FEDER-022170. A.P. wants to thank the Royal Brompton and Harefield hospital, part of the Guy’s and St Thomas’ foundation trust, London. Publisher Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland.
publishDate 2021
dc.date.none.fl_str_mv 2021-10-04T23:16:41Z
2021-08-02
2021-08-02T00:00:00Z
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
dc.type.driver.fl_str_mv info:eu-repo/semantics/article
format article
status_str publishedVersion
dc.identifier.uri.fl_str_mv http://hdl.handle.net/10362/125607
url http://hdl.handle.net/10362/125607
dc.language.iso.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv 1661-6596
PURE: 33427504
https://doi.org/10.3390/ijms22168361
dc.rights.driver.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.source.none.fl_str_mv reponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
instacron:RCAAP
instname_str Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
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reponame_str Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
collection Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
repository.name.fl_str_mv Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
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